Journal of Obstetrics and Gynaecology

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Endosalpingiosis presenting as multiple pelvic masses L. Partyka, M. Steinhoff & A. P. Lourenco To cite this article: L. Partyka, M. Steinhoff & A. P. Lourenco (2014) Endosalpingiosis presenting as multiple pelvic masses, Journal of Obstetrics and Gynaecology, 34:3, 279-281 To link to this article: http://dx.doi.org/10.3109/01443615.2013.859239

Published online: 31 Jan 2014.

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Date: 05 November 2015, At: 14:05

Journal of Obstetrics and Gynaecology 2014.34:279-281.

Gynaecology Case Reports 279 As discussed above, EMP can progress to carcinoma, particularly in postmenopausal women (Kassab et al. 2008). Giordano et al. (2007) suggested that postmenopausal status, hypertension and obesity are risk factors for malignant transformation in EMPs. In our case, the patient was neither old nor obese and did not have any evidence of hypertension. Mittal and Da Costa (2008) described the need for hysterectomy in postmenopausal women with complex hyperplasia or adenocarcinoma in EMP, even if the lesion is confined to the polyp. Although the patient in our case was young, hysterectomy could have been considered if she no longer wished to bear children. APA-LMP was also considered. However, the lesion was found in both the EMP and the surrounding endometrium. Since APA-LMP is a focal lesion, if we had chosen this diagnosis based on the polypectomy specimen, we could not have explained the lesion in the surrounding endometrium. Additionally, cellular smooth muscle was not prominent. The patient was ultimately diagnosed with an adenocarcinoma in situ arising from an EMP. We had to briefly consider the probability of a mixed tumour when we initially looked at the slides, although we could not determine if the osseous metaplasia was related to intraepithelial cancer. Pathologists should consider the possibility of misdiagnosis in cases of malignancy with osseous metaplasia, since heterologous mixed Müllerian tumours can be confined to EMPs. In our case, there was no evidence of osteosarcoma or other malignant mesenchymal tumours. Declaration of interest: The authors report no conflicts of interest. The authors alone are responsible for the content and writing of the paper.

References Arsenault AL, Ottensmeyer FP. 1984. Visualization of early intramembranous ossification by electron microscopic and spectroscopic imaging. Journal of Cell Biology 98:911–921. Costa-Paiva L, Godoy CE Jr, Antunes A Jr. 2011. Risk of malignancy in endometrial polyps in premenopausal and postmenopausal women according to clinicopathologic characteristics. Menopause 18:1278–1282. DeWaay DJ, Syrop CH, Nygaard IE. 2002. Natural history of uterine polyps and leiomyomata. Obstetrics and Gynecology 100:3–7. Giordano G, Gnetti L, Merisio C. 2007. Postmenopausal status, hypertension and obesity as risk factors for malignant transformation in endometrial polyps. Maturitas 56:190–197. Kassab A, Trotter P, Fox R. 2008. Risk of cancer in symptomatic postmenopausal women with endometrial polyps at scan. Journal of Obstetrics and Gynaecology 28:522–525. Mittal K, Da Costa D. 2008. Endometrial hyperplasia and carcinoma in endometrial polyps: clinicopathologic and follow-up findings. International Journal of Gynecological Pathology 27:45–48. Parente RC, Patriarca MT, de Moura Neto RS. 2009. Genetic analysis of the cause of endometrial osseous metaplasia. Obstetrics and Gynecology 114:1103–1108. Polat I, Sahin O, Yildirim G. 2011. Osseous metaplasia of the cervix and endometrium: a case of secondary infertility. Fertility and Sterility 95:2434.e1–e4. Tyagi SP, Saxena K, Rizvi R. 1979. Foetal remnants in the uterus and their relation to other uterine heterotopia. Histopathology 3:339–345.

Introduction Endosalpingiosis is a rare disorder of Müllerian origin, in which non-neoplastic tubal epithelium is present in ectopic anatomic locations (Hesseling and De Wilde 2000). An atypical clinical subtype, termed florid cystic endosalpingiosis, can present as multiple cystic masses that may mimic cystic ovarian neoplasms or leiomyomas (Heatley and Russell 2001; Rosenberg et al. 2011). Radiologic description of this entity is sparse and mostly limited to cases of single lesions with a dominant focus on sonography (Taneja et al. 2010; Rosenberg et al. 2011). We report a case of florid cystic endosalpingiosis presenting as multiple cystic masses along the serosal surface of the uterus and cervix, mimicking tumour recurrence in a patient with history of serous cystadenoma resection. Multimodality imaging is presented.

Case report A 55-year-old G3P1 woman presented to her primary care physician with a vague history of lower abdominal pain. Her past medical history was significant for uterine fibroids, endometriosis as seen on diagnostic laparoscopy for infertility evaluation, as well as an ovarian serous cystadenoma, for which she underwent a laparoscopic right oophorectomy, 9 years prior. Physical examination revealed an enlarged globular uterus, which had increased in size over time. The patient was referred for sonographic evaluation of the pelvis. Transabdominal and transvaginal ultrasound was performed and revealed multiple cystic lesions within the right adnexa, without detectable flow on colour Doppler (Figure 1). The patient underwent a contrast-enhanced computed tomography (CT) scan, which revealed multiple cystic lesions along the surface of the uterine body, fundus, and within the right adnexa (Figure 2). A recurrent cystic ovarian neoplasm was strongly suspected. Laboratory testing for ovarian tumour markers, including CA125 and HE-4 was performed and did not support a malignant diagnosis (CA125: 16 unit/ml [normal ⬍ 21] and

Endosalpingiosis presenting as multiple pelvic masses L. Partyka1, M. Steinhoff2 & A. P. Lourenco1 1Department of Radiology, Rhode Island Hospital and 2Department of Pathology, Women and Infants’ Hospital, Alpert Medical School, Brown University, Providence, Rhode Island, USA

DOI: 10.3109/01443615.2013.859239 Correspondence: L. Partyka, Department of Radiology, Rhode Island Hospital, Alpert Medical School, Brown University, 539 Eddy Street, Providence, Rhode Island 02903, USA. E-mail: [email protected]

Figure 1. (a) Grey scale and (b) color Doppler ultrasound images of the right adnexa showing multiple cystic structures of varying sizes without detectable blood flow on colour Doppler.

Gynaecology Case Reports

Journal of Obstetrics and Gynaecology 2014.34:279-281.

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Figure 3. Sagittal (a) and coronal (b) T2-weighted MR images showing multiple T2 hyperintense masses of varying sizes along the posterior surface of the uterus and cervix, as well as a dominant mass in the right adnexa.

benign fibrous-walled cysts with a serous epithelial lining, consistent with endosalpingiosis. There was no evidence of malignancy on microscopic evaluation of either specimen (Figure 5).

Discussion Endosalpingiosis is a rare disorder of Müllerian origin, in which a proliferation of non-neoplastic tubal epithelium is present in ectopic anatomic locations (Hesseling and De Wilde 2000). Chronic inflammation and papillary tubal hyperplasia have been proposed as a common precursor of both endosalpingiosis and all low-grade serous proliferations (Kurman et al. 2011). Endosalpingiosis is most commonly an incidental finding on post-surgical microscopic

Figure 2. Contrast-enhanced CT images of the pelvis in (a) axial, (b) coronal, and (c) sagittal planes, showing multiple cystic masses along the surface of the uterus with a dominant mass in the right adnexa. Masses ranged in size from subcentimeters to 5.5 cm.

HE 4: 62 pmol/l [normal ⬍ 115]). Further imaging with magnetic resonance imaging (MRI) was subsequently performed. MRI demonstrated the presence of multiple T2 hyperintense lesions along the serosal surface of the uterus and cervix (Figures 3 and 4), ranging in size from subcentimeters to 5.5 cm. No enhancing solid masses were visualised. A diagnosis of benign endosalpingiosis was considered. A uterine serosal biopsy of two nodular masses was performed and demonstrated

Figure 4. Post-contrast T1-weighted MR image in the axial plane showing only rim enhancement of the multiple cystic masses along the uterine surface.

Endosalpingiosis presenting as multiple pelvic masses.

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